Mutations in CEP120 cause Joubert syndrome as well as complex ciliopathy phenotypes.

PubWeight™: 0.77‹?›

🔗 View Article (PMID 27208211)

Published in J Med Genet on May 06, 2016

Authors

Susanne Roosing1, Marta Romani2, Mala Isrie3, Rasim Ozgur Rosti4, Alessia Micalizzi5, Damir Musaev4, Tommaso Mazza2, Lihadh Al-Gazali6, Umut Altunoglu7, Eugen Boltshauser8, Stefano D'Arrigo9, Bart De Keersmaecker10, Hülya Kayserili11, Sarah Brandenberger12, Ichraf Kraoua13, Paul R Mark12, Trudy McKanna12, Joachim Van Keirsbilck14, Philippe Moerman15, Andrea Poretti16, Ratna Puri17, Hilde Van Esch3, Joseph G Gleeson18, Enza Maria Valente19

Author Affiliations

1: Laboratory for Pediatric Brain Disease, New York Genome Center, Howard Hughes Medical Institute, The Rockefeller University, New York, New York, USA.
2: IRCCS Casa Sollievo della Sofferenza, Mendel Institute, San Giovanni Rotondo, Italy.
3: Department of Human Genetics, Laboratory for the Genetics of Cognition, Center for Human Genetics, KU Leuven, Belgium.
4: Department of Neurosciences, University of California San Diego (UCSD), La Jolla, California, USA.
5: IRCCS Casa Sollievo della Sofferenza, Mendel Institute, San Giovanni Rotondo, Italy Department of Biological and Environmental Science, University of Messina, Messina, Italy.
6: Department of Pediatrics, College of Medicine and Health Sciences, United Arab Emirates University, Al Ain, Abu Dhabi, United Arab Emirates.
7: Medical Genetics Department, İstanbul Medical Faculty, İstanbul University, İstanbul, Turkey.
8: Division of Pediatric Neurology, University Children's Hospital, Zurich, Switzerland.
9: Developmental Neurology Division, Fondazione IRCCS Istituto Neurologico Carlo Besta, Milan, Italy.
10: Department of Obstetrics and Gynecology, University Hospitals Leuven, Leuven, Belgium Department of Obstetrics and Gynecology, AZ Groeninge, Kortrijk, Belgium.
11: Medical Genetics Department, Koç University School of Medicine (KUSOM), Istanbul, Turkey.
12: Spectrum Health Medical Genetics, Grand Rapids, Michigan, USA.
13: Department of Child and Adolescent Neurology, National Institute Mongi Ben Hmida of Neurology of Tunis, La Rabta, Tunisia.
14: Department of Obstetrics and Gynecology, University Hospitals Leuven, Leuven, Belgium.
15: Department of Pathology, University Hospitals Leuven, Leuven, Belgium.
16: Division of Pediatric Neurology, University Children's Hospital, Zurich, Switzerland Section of Pediatric Neuroradiology, Division of Pediatric Radiology, The Russell H. Morgan Department of Radiology and Radiological Science, The Johns Hopkins School of Medicine, Baltimore, Maryland, USA.
17: Center of Medical Genetics, Sir Ganga Ram Hospital, Rajinder Nagar, New Delhi, India.
18: Laboratory for Pediatric Brain Disease, New York Genome Center, Howard Hughes Medical Institute, The Rockefeller University, New York, New York, USA Department of Neurosciences, University of California San Diego (UCSD), La Jolla, California, USA Neurogenetics Laboratory, Howard Hughes Medical Institute, Chevy Chase, Maryland, USA.
19: Section of Neurosciences, Department of Medicine and Surgery, University of Salerno, Salerno, Italy.

Articles cited by this

A framework for variation discovery and genotyping using next-generation DNA sequencing data. Nat Genet (2011) 59.36

Ciliopathies. N Engl J Med (2011) 6.28

Mutations in TMEM216 perturb ciliogenesis and cause Joubert, Meckel and related syndromes. Nat Genet (2010) 3.66

Mutations in the AHI1 gene, encoding jouberin, cause Joubert syndrome with cortical polymicrogyria. Am J Hum Genet (2004) 2.76

Cep120 and TACCs control interkinetic nuclear migration and the neural progenitor pool. Neuron (2007) 2.53

The Meckel syndrome: clinicopathological findings in 67 patients. Am J Med Genet (1984) 2.06

Joubert syndrome and related disorders: spectrum of neuroimaging findings in 75 patients. AJNR Am J Neuroradiol (2011) 1.92

AHI1 is required for photoreceptor outer segment development and is a modifier for retinal degeneration in nephronophthisis. Nat Genet (2010) 1.89

Joubert syndrome: congenital cerebellar ataxia with the molar tooth. Lancet Neurol (2013) 1.83

Joubert syndrome: insights into brain development, cilium biology, and complex disease. Semin Pediatr Neurol (2009) 1.48

MKS3/TMEM67 mutations are a major cause of COACH Syndrome, a Joubert Syndrome related disorder with liver involvement. Hum Mutat (2009) 1.43

Clinical, molecular, and genotype-phenotype correlation studies from 25 cases of oral-facial-digital syndrome type 1: a French and Belgian collaborative study. J Med Genet (2006) 1.42

Uncommon syndromes of cerebellar vermis aplasia. I: Joubert syndrome. Dev Med Child Neurol (1978) 1.39

Novel TMEM67 mutations and genotype-phenotype correlates in meckelin-related ciliopathies. Hum Mutat (2010) 1.32

Centrosome motility is essential for initial axon formation in the neocortex. J Neurosci (2010) 1.29

TCTN3 mutations cause Mohr-Majewski syndrome. Am J Hum Genet (2012) 1.29

Cep120 is asymmetrically localized to the daughter centriole and is essential for centriole assembly. J Cell Biol (2010) 1.29

C5orf42 is the major gene responsible for OFD syndrome type VI. Hum Genet (2013) 1.24

Delineation and diagnostic criteria of Oral-Facial-Digital Syndrome type VI. Orphanet J Rare Dis (2012) 1.15

Joubert syndrome: a model for untangling recessive disorders with extreme genetic heterogeneity. J Med Genet (2015) 1.14

Functional genome-wide siRNA screen identifies KIAA0586 as mutated in Joubert syndrome. Elife (2015) 1.12

Genomic analysis of Meckel-Gruber syndrome in Arabs reveals marked genetic heterogeneity and novel candidate genes. Eur J Hum Genet (2012) 1.11

CEP120 interacts with CPAP and positively regulates centriole elongation. J Cell Biol (2013) 1.09

Mutations in CSPP1 cause primary cilia abnormalities and Joubert syndrome with or without Jeune asphyxiating thoracic dystrophy. Am J Hum Genet (2013) 1.07

Uncommon posterior cranial fossa anomalies: MRI with clinical correlation. Neuroradiology (1995) 0.99

Mutations in CSPP1 lead to classical Joubert syndrome. Am J Hum Genet (2013) 0.98

Meckel-Gruber syndrome and the role of primary cilia in kidney, skeleton, and central nervous system development. Organogenesis (2013) 0.98

Co-occurrence of Joubert syndrome and Jeune asphyxiating thoracic dystrophy. Am J Med Genet A (2010) 0.96

Mutations in CSPP1, encoding a core centrosomal protein, cause a range of ciliopathy phenotypes in humans. Am J Hum Genet (2013) 0.95

A founder CEP120 mutation in Jeune asphyxiating thoracic dystrophy expands the role of centriolar proteins in skeletal ciliopathies. Hum Mol Genet (2014) 0.92

Mutations in KIAA0586 Cause Lethal Ciliopathies Ranging from a Hydrolethalus Phenotype to Short-Rib Polydactyly Syndrome. Am J Hum Genet (2015) 0.92

Oral-facial-digital syndrome type VI: is C5orf42 really the major gene? Hum Genet (2014) 0.90

Inverse cerebellum morphogenetically related to Dandy-Walker and Arnold-Chiari syndromes: bizarre malformed brain with occipital encephalocele. Johns Hopkins Med J (1972) 0.89

Tectocerebellar dysraphia and occipital encephalocele: an unusual association with abdominal situs inversus and congenital heart disease. Indian J Pediatr (2008) 0.89

KIAA0586 is Mutated in Joubert Syndrome. Hum Mutat (2015) 0.88

Mutations in human homologue of chicken talpid3 gene (KIAA0586) cause a hybrid ciliopathy with overlapping features of Jeune and Joubert syndromes. J Med Genet (2015) 0.86

Occipital extra- and intracranial lipoencephalocele associated with tectocerebellar dysraphia. Childs Nerv Syst (2004) 0.86

Talpid3-binding centrosomal protein Cep120 is required for centriole duplication and proliferation of cerebellar granule neuron progenitors. PLoS One (2014) 0.85

Tecto-cerebellar dysraphism with occipital encephalocele: not a distinct disorder, but part of the Joubert syndrome spectrum? Neuropediatrics (2011) 0.84

CNS involvement in OFD1 syndrome: a clinical, molecular, and neuroimaging study. Orphanet J Rare Dis (2014) 0.84

Tectocerebellar dysraphism with vermian encephalocele. J Child Neurol (2010) 0.82

TALPID3 controls centrosome and cell polarity and the human ortholog KIAA0586 is mutated in Joubert syndrome (JBTS23). Elife (2015) 0.80

New concepts on posterior fossa malformations. Pediatr Radiol (2008) 0.77

Pretectal pseudobobbing associated with an expanding posterior fossa cyst in tectocerebellar dysraphia: an electro-oculographic study. J Neurol (1999) 0.77

Articles by these authors

The molar tooth sign is pathognomonic for Joubert syndrome! Pediatr Neurol (2013) 1.56

Nationwide survey of Arima syndrome: a note of doubt. Brain Dev (2013) 1.38

Functional genome-wide siRNA screen identifies KIAA0586 as mutated in Joubert syndrome. Elife (2015) 1.12

Biallelic mutations in SNX14 cause a syndromic form of cerebellar atrophy and lysosome-autophagosome dysfunction. Nat Genet (2015) 1.05

Differential diagnosis of ventriculomegaly and brainstem kinking on fetal MRI. Brain Dev (2015) 0.84

Congenital brain abnormalities: an update on malformations of cortical development and infratentorial malformations. Semin Neurol (2014) 0.84

Somatic copy number alterations predict response to platinum therapy in epithelial ovarian cancer. Gynecol Oncol (2014) 0.83

Biallelic mutations in the 3' exonuclease TOE1 cause pontocerebellar hypoplasia and uncover a role in snRNA processing. Nat Genet (2017) 0.80

Mutation Update for Kabuki Syndrome Genes KMT2D and KDM6A and Further Delineation of X-Linked Kabuki Syndrome Subtype 2. Hum Mutat (2016) 0.78

t(15;21) translocations leading to the concurrent downregulation of RUNX1 and its transcription factor partner genes SIN3A and TCF12 in myeloid disorders. Mol Cancer (2015) 0.77

Overlapping SETBP1 gain-of-function mutations in Schinzel-Giedion syndrome and hematologic malignancies. PLoS Genet (2017) 0.77

Clinical and neuroimaging features as diagnostic guides in neonatal neurology diseases with cerebellar involvement. Cerebellum Ataxias (2016) 0.75

Usher syndrome type IV: clinically and molecularly confirmed by novel ARSG variants. Hum Genet (2022) 0.75

Cognitive rehabilitation in a child with Joubert Syndrome: Developmental trends and adaptive changes in a single case report. Res Dev Disabil (2015) 0.75